Patient Preference and Adherence Dovepress open access to scientific and medical research Open Access Full Text Article ORIGINAL RESEARCH Perception of Stigma in Patients with Neuromyelitis Optica Spectrum Disorder This article was published in the following Dove Press journal: Patient Preference and Adherence downloaded from https://www.dovepress.com/ on 11-Apr-2023 Patient Preference and Adherence Jose E Meca-Lallana,1 Background: Perception of stigma was associated with low self-esteem, psychological Daniel Prefasi,2 Francisco Pérez- problems, and decreased health-seeking behavior among patients with different neurological Miralles, 3 Lucía Forero, 4 disorders. The purpose of this study was to assess stigmatization and its impact in patients María Sepúlveda,5 Carmen Calles,6 with neuromyelitis optica spectrum disorder (NMOSD). María L Martínez-Ginés,7 Methods: A non-interventional study was conducted at thirteen neuroimmunology clinics in Inés González-Suárez, 8 Spain. Patients with a diagnosis of NMOSD (2015 Wingerchuk criteria) were included. The Sabas Boyero, 9 Lucía Romero- For personal use only. 8-item Stigma Scale for Chronic Illness (SSCI-8), the Expanded Disability Status Scale Pinel,10 Ángel P Sempere,11 (EDSS), the 29-item Multiple Sclerosis Impact Scale (MSIS-29), the Beck Depression Virginia Meca-Lallana,12 Inventory-Fast Screen (BDI-FS), the MOS Pain Effects Scale (MOS-PES) and the Fatigue Luis Querol,13 Lucienne Costa- Impact Scale for Daily Use (D-FIS) were used to assess the perception of stigma, disability, Frossard,14 Hugo de Castro- quality of life, mood, pain, and fatigue, respectively. Associations between outcome mea­ Trapiello, 2 Neus Canal,15 sures were analyzed using Spearman’s rank correlation. Jorge Maurino 2 Results: Seventy-one patients were studied (mean age: 47.4 years ± 14.9, 81.7% female, 1 Clinical Neuroimmunology Unit and Multiple mean time since disease onset: 9.9 years ± 8.1). The median EDSS score was 3.0 (inter­ Sclerosis CSUR. Department of Neurology. quartile range 1.5, 4.5). Stigma prevalence was 61.4% (n=43). Thirty-one patients (43.6%) Hospital Universitario “Virgen de la Arrixaca”, IMIB-Arrixaca, Murcia, Spain; 2Medical had depression. The SSCI-8 score showed a significant correlation with both physical Department, Roche Farma, Madrid, Spain; 3Unit (rho=0.576, p<0.0001) and psychological (rho=0.608, p<0.0001) MSIS-29 scales scores, of Neuroimmunology, Department of Neurology, Hospital Universitari i Politècnic La EDSS score (rho=0.349, p=0.0033), BDI-FS score (rho= 0.613, p<0.0001), MOS-PES Fe, Valencia, Spain; 4Department of Neurology, score (rho= 0.457, p<0.0001), and D-FIS score (rho=0.556, p<0.0001). Hospital Universitario Puerta del Mar, Cádiz, Conclusion: Stigma is a common phenomenon affecting over 6 out of 10 patients with Spain; 5Department of Neurology, Hospital Clínic i Provincial de Barcelona, Barcelona, NMOSD. Understanding stigma may be useful to develop educational strategies improving Spain; 6Department of Neurology, Hospital NMOSD knowledge. Universitari Son Espases, Palma de Mallorca, Spain; 7Department of Neurology, Hospital Keywords: neuromyelitis optica spectrum disorder, stigma, quality of life, depression, Universitario Gregorio Marañón, Madrid, Spain; patient-reported outcomes 8 Department of Neurology, Hospital Universitario Álvaro Cunqueiro, Vigo, Spain; 9 Department of Neurology, Hospital Universitario Cruces, Bilbao, Spain; 10 Department of Neurology, Hospital Universitari de Bellvitge, Barcelona, Spain; Introduction 11 Department of Neurology, Hospital General Neuromyelitis optica is a rare chronic inflammatory central nervous system disease Universitario de Alicante, Alicante, Spain; 12 Department of Neurology, Hospital characterized by monophasic or recurrent attacks of optic neuritis and transverse Universitario La Princesa, Madrid, Spain; 13 Department of Neurology, Hospital de la myelitis with poor or no recovery.1–3 The term neuromyelitis optica spectrum Santa Creu i Sant Pau, Barcelona, Spain; disorder (NMOSD) was established by an international panel in 2015 to enable 14 Department of Neurology, Hospital Universitario Ramón y Cajal, Madrid, Spain; early diagnosis in patients with or without aquaporin-4 antibodies.1 Motor and 15 Department of Statistics, IQVIA, Barcelona, Spain visual impairment, pain, and fatigue are among the most prevalent and debilitating symptoms.4,5 Residual disability usually limit daily function and independence over Correspondence: Jorge Maurino Roche Farma, Ribera Del Loira, 50, time.6,7 Madrid, 28042, Spain Tel +34 913 24 81 00 The definition of stigma traditionally refers to a feeling of disapproval that Email jorge.maurino@roche.com society has about something, especially when this is unfair.8 Perception of stigma submit your manuscript | www.dovepress.com Patient Preference and Adherence 2021:15 713–719 713 DovePress © 2021 Meca-Lallana et al. This work is published and licensed by Dove Medical Press Limited. The full terms of this license are available at https://www.dovepress.com/ http://doi.org/10.2147/PPA.S305707 terms.php and incorporate the Creative Commons Attribution – Non Commercial (unported, v3.0) License (http://creativecommons.org/licenses/by-nc/3.0/). By accessing the work you hereby accept the Terms. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed. For permission for commercial use of this work, please see paragraphs 4.2 and 5 of our Terms (https://www.dovepress.com/terms.php). Meca-Lallana et al Dovepress was a common phenomenon among people with neurolo­ from 1 (never) to 5 (always). Total scores range from 8 gical disorders in a survey recently conducted by the to 40, with a cut-off score >8 indicating the presence of European Federation of Neurological Associations.8 The stigmatization. The MSIS-29 is a self-report question­ impact of stigma has been documented across a number of naire used to determine the impact of MS on health- neurological conditions contributing to lack of confidence, related quality of life.27 It consists of two composite depression, anxiety, decreased health-seeking behavior, domains including physical (20 items) and psychologi­ and unequal life opportunities.8–12 cal impacts (9 items). Items are rated using four-point There is limited information available on the presence response categories: not at all, a little, moderately, and of stigma in patients with neuroinflammatory and demye­ extremely. Scores on the physical and psychological linating disorders. Perception of stigma has been recently impact subscales can range from 20 to 80 and from 9 explored in multiple sclerosis (MS).13–17 Different studies to 36, respectively. Higher scores indicate greater showed a prevalence of perceived stigma of 20 and 80% in impact. The BDI-FS is a seven-item questionnaire asses­ patients with relapsing-remitting and primary progressive sing the level of depressive symptoms.28 Responses to MS, respectively.16,17 Even in patients with low physical the items are provided on a four-point scale (no symp­ disability, stigma was associated with poor quality of life, toms to severe symptoms) with total score ranging from depression, and unemployment.15,17,18 0 to 21. Cut-off scores ≥4 and ≥9 are used to define the Depression and psychological distress affect subjective presence of depression and moderate-to-severe depres­ well-being in patients with NMOSD.19–22 However, no sion, respectively. The MOS-PES is a validated, six- studies have focused on stigma and NMOSD. The purpose item, self-report questionnaire to assess how pain and of this study was to assess stigmatization and its impact unpleasant sensations interfere with mood, ability to on patients living with NMOSD. walk or move, sleep, work, recreation, and enjoyment of life.29 Total score ranges from 6 to 30, with higher Methods scores indicating greater impact of pain. The D-FIS is A non-interventional, cross-sectional study was conducted an eight-item, self-report instrument designed to mea­ at thirteen hospital-based neuroimmunology clinics in sure subjective daily experience of fatigue.30 Items are Spain (PERSPECTIVES-NMO study). Eligibility criteria rated using a 5-point Likert scale from 1 (never) to 5 included being at least 18 years old and having a diagnosis (always). Higher scores indicate greater fatigue of NMOSD according to Wingerchuk 2015 criteria.23 This perception. study was conducted in accordance with the Good Clinical Practice Guidelines of the International Conference on Statistical Analysis Harmonisation and with the ethical principles of the Associations between outcome measures were analyzed Declaration of Helsinki and was approved by the investi­ using Spearman’s rank correlation. Logistic regression gational review board of Galicia (CEIm-G), Santiago de was used to assess the contribution of stigma to symptoms Compostela, Spain. Written informed consent was of depression (depressed, not depressed). obtained from all participants. Results Outcome Measures A total of 71 patients were included in the study. The mean The 8-item Stigma Scale for Chronic Illness (SSCI-8), the (± SD) age was 47.4 ± 14.9 years and 81.7% were female. Expanded Disability Status Scale (EDSS), the 29-item The mean time since disease onset was 9.9 years ± 8.1 and Multiple Sclerosis Impact Scale (MSIS-29), the Beck the median EDSS score was 3.0 (interquartile range 1.5, Depression Inventory-Fast Screen (BDI-FS), the MOS 4.5). Thirty-one patients (43.6%) had depression. Pain Effects Scale (MOS-PES) and the Fatigue Impact Demographic and clinical characteristics of the sample Scale for Daily Use (D-FIS) were used to assess the are shown in Table 1. perception of stigma, disability, quality of life, mood, The prevalence of stigma was 61.4% (95% CI 46.9, pain and fatigue, respectively.24–30 76.0 [n=43]) (Table 2). Feeling of being left out of things, The SSCI-8 was developed to assess perceptions of followed by embarrassment because of physical limita­ stigma across different neurological conditions.24,25 It tions and people avoided me because of my illness were comprises eight items rated on a 5-point Likert scale the items with the highest impact. 714 submit your manuscript | www.dovepress.com Patient Preference and Adherence 2021:15 DovePress Powered by TCPDF (www.tcpdf.org) Dovepress Meca-Lallana et al Table 1 Demographic and Clinical Characteristics Discussion Total Little is known about the patient´s experience of living (n=71) with NMOSD.31 Fear of disability and impaired activ­ Age (years), mean ± SD 47.4 ± 14.9 ities of daily living were associated with uncertainty about the disease trajectory.31,32 Limitations in social Sex (female), n (%) 57 (80.3) activities and type of work were reported by most of Education (university), n (%) 30 (42.2) 522 patients participating in the US online community Living with family members or a partner, n (%) 63 (90) PatientsLikeMe.4 Somatic complaints and hopelessness were correlated with disease duration.22 Barzegar et al Employment status (part or full-time employed), 21 (30) n (%) found that NMOSD patients had significantly worse quality of life compared with MS patients, with fatigue Disease duration (years), mean ± SD 9.9 ± 8.1 being the most important variable for predicting Onset attack type, n (%) variance.33 Myelitis 34 (47.8) The impact of stigma has been documented across Optic neuritis 27 (38) a number of neurological disorders, especially Myelitis + optic neuritis 3 (4.2) Parkinson´s disease, epilepsy, and Alzheimer´s Other 7 (9.8) disease.8,10 Stigma was a common phenomenon in Relapsing form, n (%) 59 (83.1) a survey with 1,373 patients with different neurological Number of relapses since diagnosis, mean ± SD 2.9 ± 2.3 conditions from 37 countries.8 Lack of understanding Number of relapses in the last year, mean ± SD 0.5 ± 0.9 was seen as the biggest cause of this, followed by mis­ conceptions about these disorders and their hidden nat­ Coexisting autoimmune disease, n (%) 21 (29.6) ure. More than thirty percent of participants reported EDSS score, median (IQR) 3.0 (1.5, 4.5) regularly being made to feel that their condition is their Physical impact MSIS-29 score, mean ± SD 41.9 ± 16.7 fault, over twenty percent felt that people avoid them often or always, and forty-five percent felt that they are Psychological impact MSIS-29 score, mean ± SD 20.9 ± 8.3 regularly left out of things. MOS-PES score, mean ± SD 13.9 ± 6.3 Perception of stigma in patients with NMOSD using D-FIS score, mean ± SD 9.0 ± 8.3 standardized measures has not been previously explored. In our study, more than sixty percent of patients experi­ BDI-FS score, mean ± SD 3.6 ± 3.4 enced stigmatization. Stigma was found to have a strong Patients with depression, n (%) 31 (43.6) negative correlation with physical and psychological qual­ Patients with moderate to severe depression, n (%) 8 (11.2) ity of life. Abbreviations: BDI-FS, Beck Depression Inventory-Fast Screen; D-FIS, Fatigue There is scarce information regarding psychiatric Impact Scale for Daily Use; EDSS, Expanded Disability Status Scale; IQR, interquar­ comorbidities in patients with NMOSD. Based on tile range; MOS-PES, Pain Effects Scale; MSIS-29, 29-item Multiple Sclerosis Impact Scale; SD, standard deviation. a retrospective analysis of a US administrative claims data­ base, the prevalence of depression in patients with NMO (n=1,349) and highly active NMO (n=134; defined as at least two relapses within twelve months of the patient’s first The SSCI-8 score showed a significant correlation NMO encounter in the database) was estimated at 17.9% with physical and psychological MSIS-29 scores and 25.4%, respectively.34 Fernandez et al found that ten (rho=0.576 and 0.608, p<0.0001, respectively), EDSS patients with NMOSD (50%) had current depressive symp­ score (rho=0.349, p=0.0033), BDI-FS score (rho= toms versus five (28%) patients with MS (p=0.16).35 Six 0.613, p<0.0001), MOS-PES score (rho= 0.457, (30%) patients with NMOSD versus only one (5.5%) p<0.0001), and D-FIS score (rho=0.556, p<0.0001) patient with MS had attempted suicide at least once (Table 3). Stigma was found to positively predict con­ (p=0.05). Current suicide risk was high in patients with current depression (OR=1.32; 95% CI: 1.13–1.55, NMOSD (8, 40%) and moderate in patients with MS (4, p=0.0004). 22%). In our study, thirty-one patients (43.6%) had submit your manuscript | www.dovepress.com Patient Preference and Adherence 2021:15 715 DovePress Powered by TCPDF (www.tcpdf.org) Meca-Lallana et al Dovepress Table 2 Perception of Stigma n=70 SSCI-8 score, median (IQR) 10 (8.0, 15.0) Stigma prevalence (total score >8), n (%) 43 (61.4) Frequency, n (%) SSCI-8 Items Never/Rarely Sometimes Often/Always Because of my illness, some people seemed uncomfortable with me 60 (85.7) 9 (12.9) 1 (1.4) Because of my illness, some people avoided me 57 (81.4) 8 (11.4) 5 (7.1) Because of my illness, I felt left out of things 52 (74.3) 10 (14.3) 8 (11.4) Because of my illness, people were unkind to me 61 (87.1) 7 (10) 2 (2.9) Because of my illness, people avoided looking at me 62 (88.6) 6 (8.6) 2 (2.9) I felt embarrassed about my illness 64 (91.4) 4 (5.7) 2 (2.9) I felt embarrassed because of my physical limitations 52 (74.3) 13 (18.6) 5 (7.1) Some people acted as though it was my fault I have this illness 61 (87.1) 5 (7.1) 4 (5.7) Notes: ©2008–2019 David Cella on behalf of the National Institute for Neurological Disorders and Stroke (NINDS); some content used with permission of the PROMIS Health Organization. Abbreviations: SSCI-8, 8-item Stigma Scale for Chronic Illness; IQR, interquartile range. depression. Differences regarding our findings could be depression (OR=1.21, p=0.007) and moderate-to-severe based on cultural factors and the administration of different depression (OR=1.37, p=0.003).36 instruments to assess depressive symptoms across studies. Patients with rare diseases show even worse quality of Stigma is a previously studied predictor of depres­ life compared to the general population and people with sion in patients with MS. Cadden et al and Pérez- common chronic diseases.37 A comprehensive disease Miralles et al assessed the association between stigma information and educational efforts to achieve an adequate and depression in MS, concurrently and public knowledge of NMOSD may help in decreasing its 15,36 prospectively. A moderate bivariate correlation stigma. The ultimate goal of all such initiatives should be between stigma and depression measured at baseline to prevent the isolation of NMOSD patients and their and one year later was found in a survey with 5,413 families and to facilitate their social integration. participants administered by the North American The relatively small sample size and cross-sectional Research Committee on Multiple Sclerosis.15 Almost design are the main limitations of this study. A causal eighty percent of a sample of 55 patients with primary relationship between stigma and depression cannot be progressive MS reported some degree of stigmatization established. We also cannot rule out the possibility that at baseline and one year later.16,36 Increased baseline the perception of stigma was influenced by other fac­ SSCI-8 scores were associated with a higher risk of tors that have not been analyzed in this study, such as Table 3 Correlations Between SSCI-8 and BDI-FS, MOS PES, D-FIS, and EDSS Physical MSIS-29 Psychological BDI-FS MOS-PES D-FIS EDSS MSIS-29 SSCI-8 r 0.576 0.608 0.613 0.457 0.556 0.349 p <0.0001 <0.0001 <0.0001 <0.0001 <0.0001 0.0033 Abbreviations: BDI-FS, Beck Depression Inventory-Fast Screen; D-FIS, Fatigue Impact Scale for Daily Use; EDSS, Expanded Disability Status Scale; MOS-PES, Pain Effects Scale; MSIS-29, 29-item Multiple Sclerosis Impact Scale; r, Spearman’s rank correlation coefficient; SSCI-8, 8-item Stigma Scale for Chronic Illness. 716 submit your manuscript | www.dovepress.com Patient Preference and Adherence 2021:15 DovePress Powered by TCPDF (www.tcpdf.org) Dovepress Meca-Lallana et al current immunosuppressant drugs and disease- Disclosure modifying therapies for the management of NMOSD, D. Prefasi, H. de Castro-Trapiello, and J. Maurino are impact of medical or psychological comorbid condi­ employees of Roche Farma Spain. N. Canal is an employee tions, or serologic status. However, the study sample of IQVIA Spain and IQVIA has received honoraria from was managed at 13 different hospitals throughout Roche for clinical research services. F. Pérez-Miralles has Spain, allowing results to be generalized to clinical received compensation for serving on scientific advisory practice. boards or speaking honoraria from Almirall, Biogen Idec, Genzyme, Merck-Serono, Mylan, Novartis, Roche, Sanofi- Conclusion Aventis, and Teva, outside the submitted work. M. Sepúlveda Perception of stigma is a common phenomenon in reports personal fees from Roche and UCB; travel reimbur­ NMOSD, even in a clinically stable population with low sement from Sanofi and Zambon. C. Calles reports personal physical disability. Further studies with longitudinal fol­ fees from Biogen, Sanofi, Merck, Novartis, Teva, and Roche, low-up are needed to confirm the relationships between outside the submitted work. S. Boyero reports personal fees stigma and quality of life and mood and elucidate the from Roche, during the conduct of the study. L. Romero- underlying mechanisms. Pinel took part in the observational study of Neuromyelitis optica: perspective NMO-ML41397. She also received hon­ oraria compensation to participate in advisory boards, colla­ Data Sharing Statement borations as a consultant and scientific communications and Qualified researchers may request access to individual received research support, funding for travel and congress patient-level data through the clinical study data request expenses from Biogen Idec, Bayer, Almirall, Merck, Sanofi- platform (https://vivli.org/). Further details on Roche’s Genzyme, Roche, Novartis, and Teva. She reports participa­ criteria for eligible studies are available here (https:// tion in several clinical trials. Á Sempere reports personal fees vivli.org/members/ourmembers/). For further details on from Biogen, Roche, Merck, Sanofi, and Teva, outside the Roche’s Global Policy on the Sharing of Clinical submitted work. L. Querol reports grants from ISCIII, Information and how to request access to related clinical CIBERER, GBS-CIDP Foundation International, personal study documents, see here https://www.roche.com/ fees from UCB Pharma as the global principal investigator research_and_development/who_we_are_how_we_work/ in clinical trial, grants and/or personal fees from Grifols, clinical_trials/our_commitment_to_data_sharing.htm. Roche, Merck, Akcea, Biogen, Novartis, Sanofi Genzyme, and Annexon, outside the submitted work. L. Costa-Frossard Acknowledgments reports personal fees from Roche, during the conduct of the The authors would like to acknowledge all patients and study; speaker fees, travel support, and/or served on advisory their families for making the PERSPECTIVES-NMO boards from Almirall, Bayer, Biogen, Biopass, Bristol-Myer study possible. s, Ipsen, Merck, Novartis, Sanofi, and Teva, outside the submitted work. The authors report no other conflicts of Author Contributions interest in this work. All authors made a significant contribution to the work reported, whether that is in the conception, study design, References execution, acquisition of data, analysis and interpretation, 1. Marignier R, Cobo Calvo A, Vukusic S. Neuromyelitis optica and or in all these areas; took part in drafting, revising or neuromyelitis optica spectrum disorders. Curr Opin Neurol. 2017;30 critically reviewing the article; gave final approval of the (3):208–215. doi:10.1097/WCO.0000000000000455 version to be published; have agreed on the journal to 2. Borisow N, Mori M, Kuwabara S, Scheel M, Paul F. Diagnosis and treatment of NMO spectrum disorder and MOG-encephalomyelitis. which the article has been submitted; and agree to be Front Neurol. 2018;9:888. doi:10.3389/fneur.2018.00888 accountable for all aspects of the work. 3. Papp V, Magyari M, Aktas O, et al. Worldwide incidence and pre­ valence of neuromyelitis optica: a systematic review. Neurology. 2021;96(2):59–77. doi:10.1212/WNL.0000000000011153 Funding 4. Eaneff S, Wang V, Hanger M, et al. Patient perspectives on neuro­ myelitis optica spectrum disorders: data from the PatientsLikeMe This study was funded by the Medical Department of online community. Mult Scler Relat Disord. 2017;17:116–122. Roche Farma Spain (ML41397). doi:10.1016/j.msard.2017.07.014 submit your manuscript | www.dovepress.com Patient Preference and Adherence 2021:15 717 DovePress Powered by TCPDF (www.tcpdf.org) Meca-Lallana et al Dovepress 5. Jarius S, Paul F, Weinshenker BG, et al. Neuromyelitis optica. Nat 22. Wingerchuk DM, Banwell B, Bennett JL, et al. International con­ Rev Dis Primers. 2020;6(1):85. doi:10.1038/s41572-020-0214-9 sensus diagnostic criteria for neuromyelitis optica spectrum disorders. 6. Methley AM, Mutch K, Moore P, Jacob A. Development of a Neurology. 2015;85(2):177–189. doi:10.1212/WNL.000000000 patient-centred conceptual framework of health-related quality of 0001729 life in neuromyelitis optica: a qualitative study. Health Expect. 23. Molina Y, Choi SW, Cella D, et al. The Stigma Scale for Chronic 2017;20(1):47–58. doi:10.1111/hex.12432 Illnesses 8-item version (SSCI-8): development, validation and use 7. Banerjee A, Ng J, Coleman J, Ospina JP, Mealy M, Levy M. across neurological conditions. Int J Behav Med. 2013;20:450–460. Outcomes from acute attacks of neuromyelitis optica spectrum dis­ doi:10.1007/s12529-012-9243-4 order correlate with severity of attack, age and delay to treatment. 24. Ballesteros J, Martínez-Ginés ML, García-Domínguez JM, et al. Mult Scler Relat Disord. 2019;28:60–63. doi:10.1016/j. Assessing stigma in multiple sclerosis: psychometric properties of msard.2018.12.010 the Eight-Item Stigma Scale for Chronic Illness (SSCI-8). Int J MS 8. European Federation of Neurological Associations (EFNA). Survey Care. 2019;21(5):195–199. doi:10.7224/1537-2073.2018-053 on stigma and neurological disorder; 2020. Available from: https:// 25. Kurtzke JF. Rating neurologic impairment in multiple sclerosis: an www.efna.net/wp-content/uploads/2020/07/SurveyReport2020.pdf. expanded disability status scale (EDSS). Neurology. 1983;33 Accessed April 2, 2021. (11):1444–1452. doi:10.1212/wnl.33.11.1444 9. Burgener SC, Berger B. Measuring perceived stigma in persons with 26. Hobart J, Lamping D, Fitzpatrick R, et al. The Multiple Sclerosis progressive neurological disease: Alzheimer’s dementia and Impact Scale (MSIS-29): a new patient-based outcome measure. Parkinson’s disease. Dementia. 2008;7(1):31–53. Brain. 2001;124:962–973. doi:10.1093/brain/124.5.962 10. Rao D, Choi SW, Victorson D, et al. Measuring stigma across 27. Benedict RH, Fishman I, McClellan MM, et al. Validity of the beck neurological conditions: the development of the stigma scale for depression inventory-fast screen in multiple sclerosis. Mult Scler. chronic illness (SSCI). Qual Life Res. 2009;18(5):585–595. 2003;9:393–396. doi:10.1191/1352458503ms902oa doi:10.1007/s11136-009-9475-1 28. Archibald CJ, McGrath P, Ritvo PG, et al. Pain prevalence, severity 11. Ochoa-Morales A, Fresan-Orellana A, Hernández-Mojica T, et al. and impact in a clinic sample of multiple sclerosis patients. Pain. Perceived discrimination in patients with multiple sclerosis and 1994;58(1):89–93. doi:10.1016/0304-3959(94)90188-0 depressive symptomatology. Mult Scler Relat Disord. 29. Benito-León J, Martínez-Martín P, Frades B, et al. Impact of fatigue 2020;48:102705. doi:10.1016/j.msard.2020.102705 in multiple sclerosis: the fatigue impact scale for daily use (D-FIS). 12. Elliot VL, Morgan D, Kosteniuk J, et al. Health-related stigma of Mult Scler. 2007;13(5):645–651. doi:10.1177/1352458506073528 noncommunicable neurological disease in rural adult populations: 30. Mutch K, Methley A, Moore P, et al. Life on hold: the experience of a scoping review. Health Soc Care Community. 2019;27(4):e158– living with neuromyelitis optica. Disabil Rehabil. 2014;36 e188. doi:10.1111/hsc.12694 (13):1100–1107. doi:10.3109/09638288.2013.833301 13. Anagnostouli M, Katsavos S, Artemiadis A, et al. Determinants of 31. Kitley J, Leite MI, Nakashima I, et al. Prognostic factors and disease stigma in a cohort of hellenic patients suffering from multiple sclero­ course in aquaporin-4 antibody-positive patients with neuromyelitis sis: a cross-sectional study. BMC Neurol. 2016;16:101. doi:10.1186/ optica spectrum disorder from the United Kingdom and Japan. Brain. s12883-016-0621-4 2012;135(Pt6):1834–1849. doi:10.1093/brain/aws109 14. Cadden MH, Arnett PA, Tyry TM, et al. Judgment hurts: the psycholo­ 32. Barzegar M, Badihian S, Mirmosayyeb O, et al. Comparative study gical consequences of experiencing stigma in multiple sclerosis. Soc Sci of quality of life, anxiety, depression, and fatigue among patients Med. 2018;208:158–164. doi:10.1016/j.socscimed.2018.01.015 with neuromyelitis optica spectrum disorder and multiple sclerosis: 15. Pérez-Miralles F, Prefasi D, García-Merino A, et al. Perception of the first report from Iran. Mult Scler Relat Disord. 2018;22:161–165. stigma in patients with primary progressive multiple sclerosis. Mult doi:10.1016/j.msard.2018.04.009 Scler J Exp Transl Clin. 2019;5(2):2055217319852717. doi:10.1177/ 33. Ajmera MR, Boscoe A, Mauskopf J, et al. Evaluation of comorbid­ 2055217319852717 ities and health care resource use among patients with highly active 16. Maurino J, Martínez-Ginés ML, García-Domínguez JM, et al. neuromyelitis optica. J Neurol Sci. 2018;384:96–103. doi:10.1016/j. Workplace difficulties, health-related quality of life, and perception jns.2017.11.022 of stigma from the perspective of patients with multiple sclerosis. 34. Fernández VC, Alonso N, Melamud L, et al. Psychiatric comorbid­ Mult Scler Relat Disord. 2020;41:102046. doi:10.1016/j. ities and suicidality among patients with neuromyelitis optica spec­ msard.2020.102046 trum disorders in Argentina. Mult Scler Relat Disord. 2018;19:40–43. 17. Dennison L, Yardley L, Devereux A, et al. Experiences of adjusting doi:10.1016/j.msard.2017.11.002 to early stage multiple sclerosis. J Health Psychol. 2011;16 35. Pérez-Miralles F, Prefasi D, García-Merino A, et al. Short-term data (3):478–488. doi:10.1177/1359105310384299 on disease activity, cognition, mood, stigma and employment out­ 18. Shi Z, Chen H, Lian Z, et al. Factors that impact health-related comes in a cohort of patients with primary progressive multiple quality of life in neuromyelitis optica spectrum disorder: anxiety, sclerosis (UPPMS study). Mult Scler Relat Disord. 2021;50:102860. disability, fatigue and depression. J Neuroimmunol. doi:10.1016/j.msard.2021.102860 2016;293:54–58. doi:10.1016/j.jneuroim.2016.02.011 36. Broersma F, Oeseburg B, Dijkstra J, et al. The impact of 19. Nakazawa K, Noda T, Ichikura K, et al. Resilience and depression/ self-perceived limitations, stigma and sense of coherence on quality anxiety symptoms in multiple sclerosis and neuromyelitis optica of life in multiple sclerosis patients: results of a cross-sectional study. spectrum disorder. Mult Scler Relat Disord. 2018;25:309–315. Clin Rehabil. 2018;32:536–545. doi:10.3389/fneur.2018.00888 doi:10.1016/j.msard.2018.08.023 37. Bogart KR, Irvin VL. Health-related quality of life among adults with 20. Beekman J, Keisler A, Pedraza O, et al. Neuromyelitis optica spectrum diverse rare disorders. Orphanet J Rare Dis. 2017;12(1):177. disorder: patient experience and quality of life. Neurol Neuroimmunol doi:10.1186/s13023-017-0730-1 Neuroinflamm. 2019;6(4):e580. doi:10.1212/NXI.0000000000000580 21. Shin JS, Kwon YN, Choi Y, et al. Comparison of psychiatric dis­ turbances in patients with multiple sclerosis and neuromyelitis optica. Medicine (Baltimore). 2019;98(38):e17184. doi:10.1097/ MD.0000000000017184 718 submit your manuscript | www.dovepress.com Patient Preference and Adherence 2021:15 DovePress Powered by TCPDF (www.tcpdf.org) Dovepress Meca-Lallana et al Patient Preference and Adherence Dovepress Publish your work in this journal Patient Preference and Adherence is an international, peer-reviewed, states are major areas of interest for the journal. This journal has open access journal that focusing on the growing importance of been accepted for indexing on PubMed Central. The manuscript patient preference and adherence throughout the therapeutic conti­ management system is completely online and includes a very quick nuum. Patient satisfaction, acceptability, quality of life, compliance, and fair peer-review system, which is all easy to use. 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